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Ann Rheum Dis: first published as 10.1136/ard.48.8.689 on 1 August 1989. Downloaded from

Annals of the Rheumatic 1989; 48: 689-691 Upper limb pyrophosphate outside the carpal tunnel

J C GERSTER AND R LAGIER From the and Rehabilitation Centre, University Hospital, CHUV, Lausanne and the Department of Pathology (Osteoarticular Unit), Medical School, Geneva, Switzerland

SUMMARY Three cases of calcium pyrophosphate dihydrate (CPPD) crystal deposits in sheaths outside the carpal tunnel are reported. Crystals were shown by x ray diffraction analysis in one case and by compensated light microscopy in the other two. Surgical excision of the tendon synovial sheath had to be done in two cases (one case with CPPD crystal deposits).

Extra-articular deposition of calcium pyrophosphate swollen and tender and the shoulders were limited dihydrate (CPPD) crystals has been described in and tender. Radiographs showed rare cases, especially in the ligamentum flavuml4 of both wrists, both knees, and a destructive and in various kinds of : triceps,5 flexor of the left shoulder. Degenerative digitorum,3 and Achilles tendon.6 7 It has also been reported in ,5 in subcutaneous tissue,"O and copyright. even in the dura mater.11 Presumed similar deposits have been radiologically reported in patients other- wise known for CPPD crystal deposition -for example, in transverse ligament of the second cervical vertebra12 and in flexor digitorum.13 The association of carpal tunnel syndrome with articular chondrocalcinosis has been classically

reported'4v7; http://ard.bmj.com/ deposits of CPPD crystals have been found in the volar carpal ligament as well as in the tendon sheaths. "" l9 We report the finding of CPPD crystals in the synovial fluid from tendon sheaths outside the carpal tunnel.

Case report on September 29, 2021 by guest. Protected

CASE 1 A 72 year old woman had been operated on for a bilateral carpal tunnel syndrome 10 years ago. She had been suffering from pain in the left knee for about five years and from pain in both shoulders for three years. In addition, she had had painful swelling of the ventral part of the fifth left finger for about 10 months. Clinically, the tendon sheath was diffusely swollen and tender and motion of the finger was restricted (Fig. 1). The left knee was sZ i.^, . ^ Accepted for publication 9 January 1989. Correspondence to Dr J C Gerster, Rheumatology and Rehabilita- tion Centre, University Hospital, CHUV, CH-1011 Lausanne, Fig. 1 Case 1. Diffuse swelling ofthe tendon sheath ofthe Switzerland. fifth finger. 39 Ann Rheum Dis: first published as 10.1136/ard.48.8.689 on 1 August 1989. Downloaded from

690 Gerster, Lagier changes of the first carpometacarpal were also examinatiuon was performed on stained (haemat- found in the left hand. oxylin and eosin and von Kossa stain) and unstained Examination of the synovial fluid of the left knee slides, which were examined by polarised light. It showed 0*2x109 leucocytes/l with rare intracellular showed a non-specific synovial hyperplasia with scar positive birefringent crystals. Red S alizarin staining remodelling but without CPPD crystal deposits. was negative. Synovial fluid was removed from the Examination of some fragments by transmitted tenosynovial cavity of the finger (25 January 1988); electron microscopy and by x ray electron diffraction it was gelatinous and contained rare white deposits did not show CPPD crystals. 2-3 mm in diameter. x Ray powder diffraction analyses of this material showed CPPD crystals in triclinic form. Tenosynovitis recurred, and a syno- Discussion vectomy was performed in April 1988. Histological examination was performed on slides stained by The common feature of these three cases is a hand haematoxylin and eosin and by von Kossa stain for tenosynovitis whose exudated synovial fluid con- calcium phosphate. It showed a non-specific scar tained CPPD crystals which were disclosed by x ray remodelling of the synovium with gelatinous deposits diffraction in one case and by the presence of rod- and only an isolated small area of CPPD crystal like positive birefringent crystals in the others. In deposits. Recovery was uneventful. only one case were CPPD crystal deposits found in the neighbouring synovium and this patient CASE 2 had presented with radiological signs of chondro- In February 1988 a 77 year old man developed calcinosis in both knee and wrist joints. In the other nodular thickening of the palmar surface of his left two cases chondrocalcinosis was shown by x ray wrist. He was also suffering from pain in the first examination in only one of them. carpometacarpal joints bilaterally and from chronic These cases show that CPPD crystal deposits in pain in the left knee with some acute typical attacks the hand in extra-articular locations induce not only of pseudogout. Examination of the left wrist showed a carpal tunnel syndrome but also tenosynovitiscopyright. a distended synovial sheath of the flexor carpi even in the absence of radiological signs of chondro- ulnaris 0-8 cm in diameter. calcinosis elsewhere in the joints. Synovial fluid There were no signs of carpal tunnel syndrome. examination seems to be more simple and sensitive x Ray examination showed chondrocalcinosis of than a synovial biopsy, particularly as histological both knees but no chondrocalcinosis of the wrists. changes are not equally spread. It is uncertain Aspiration of the synovial fluid from the teno- whether the primary CPPD crystal deposits involve the tenosynovial membrane or the tendon itself. synovial cavity produced a clear fluid containing http://ard.bmj.com/ 1*3 x 109 leucocytes/l (98% mononuclear) with plenty of intracellular positive rod-like birefringent crystals. The authors thank Professor C A Baud, Institute of Morphology, University Hospital, Geneva, for x ray diffractions and Dr C A topical corticosteroid agent was injected with a Simonetta, Permanence Chirurgicale de Longeraie, Lausanne, for good result. clinical data.

CASE 3 References A 53 year old woman had suffered from tenosynovitis 1 Bywaters E G L, Hamilton EB D, Williams R. The spine in of the extensor tendon sheaths of the fingers in 1965. idiopathic haemochromatosis. Ann Rheum Dis 1971; 30:453-65. on September 29, 2021 by guest. Protected A was but there was 2 Ellman M H, Vazquez T, Ferguson L, Mandel N. Calcium tenosynovectomy performed, pyrophosphate deposition in ligamentum flavum. no histological examination. In 1986 the symptoms Rheum 1978; 21: 611-3. occurred again. 3 Lagier R. L'approche anatomo-pathologique du concept de Clinical examination was normal except for a chondrocalcinose articulaire. Rhumatologie 1981; 33: 421-37. swollen tendon sheath of the extensor tendons of the 4 Nagashima C, Takahama M, Shibata T, et al. Calcium pyrophosphate dihydrate deposits in the cervical ligamenta right wrist and for a tendemess of the first carpo- flava causing myeloradiculopathy. J Neurosurg 1984; 60: 69-80. metacarpal . Examination of 0*5 ml of synovial 5 Gerster J C, Lagier R, Boivin G. related to fluid from the tendon synovial cavity showed 0*9 x 109 calcium pyrophosphate dihydrate crystal deposition disease. leucocytes/l (98% mononuclear) with intracellular Clinical and pathologic study. Arthritis Rheum 1982; 25: 989-96. 6 Gerster J C, Baud C A, Lagier R, Boussina I, Fallet G H. and extracellular positive rod-like birefringent Tendon calcifications in chondrocalcinosis. Arthritis Rheum crystals. 1977; 20: 717-22. An x ray examination of the hand and knees failed 7 Gerster J C, Lagier R, Boivin G. associated to show of chondrocalcinosis. A with chondrocalcinosis. J Rheumatol 1980; 7: 82-9. signs tenosyno- 8 Leisen J C, Austad E D, Bluhm G B, Sigler J W. The tophus in vectomy was performed in November 1986. The calcium pyrophosphate deposition disease. JAMA 1980; 244: synovial fluid contained crystals. Histological 1711-2. Ann Rheum Dis: first published as 10.1136/ard.48.8.689 on 1 August 1989. Downloaded from

Upper limb pyrophosphate tenosynovitis 691

9 Rothschild B M, Round M J. Subcutaneous crystal deposition 15 Rubinstein H M, Shah D M. Pseudogout. Semin Arthritis in pseudogout. JAMA 1980; 244: 2079-80. Rheum 1973; 2: 259-80. 10 Schumacher H R Jr, Bonner H, Thompson J J, Kester W L, 16 Spiegel P G, Ginsberg M, Skosey J L, Kwong P. Acute carpal Benner J J. Tumor-like swelling of the distal phalanx tunnel syndrome secondary to pseudogout: case report. Clin due to calcium pyrophosphate dihydrate crystal deposition. Orthop 1976; 120: 185-7. Arthritis Rheum 1984; 27: 1428-32. 17 Goodwin D R A, Arbel R. Pseudogout of the wrist presenting 11 Grahame R, Sutor D J, Mitchener M B. Crystal deposition in as acute median nerve compression. J Hand Surg [Br] 1985; 10: hyperparathyroidism. Ann Rheum Dis 1971; 30: 597-604. 261-2. 12 Dirheimer Y, Bensimon C, Christmann D, Wackenheim C. Syndesmo-odontoid joint and calcium pyrophosphate dihydrate 18 Gerster J C, Lagier R, Boivin G, Schneider C. Carpal tunnel deposition disease. Neuroradiology 1983; 25: 319-21. syndrome in chondrocalcinosis of the wrist. Clinical and 13 Bywaters E G L, Dixon A St J, Scott J. Joint lesion of histologic study. Arthritis Rheum 1980; 23: 924-31. hyperparathyroidism. Ann Rheum Dis 1963; 22: 171-87. 19 Lagier R, Boivin G, Gerster J C. Carpal tunnel syndrome 14 Weinstein J D, Dick H M, Grantham S A. Pseudogout, hyper- associated with mixed calcium pyrophosphate dihydrate and parathyroidism, and carpal tunnel syndrome: a case report. apatite crystal deposition in tendon synovial sheath. Arthritis J Bone Joint Surg [Am], 1968; 50: 1669-74. Rheum 1984; 27: 1190-5. copyright. http://ard.bmj.com/ on September 29, 2021 by guest. Protected